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经新辅助地诺单抗治疗以显示骨外病变的β-连环蛋白阳性骨巨细胞瘤伴病理性骨折

Beta-Catenin Positive Giant Cell Tumor of Bone with Pathological Fracture Treated with Neoadjuvant Denosumab Therapy for Visualizing Extraskeletal Lesions.

作者信息

Ogawa Itaru, Kaneuchi Yoichi, Hakozaki Michiyuki, Sato Shunsuke, Suzuki Takeo, Yamada Shoki, Hasegawa Osamu, Nikaido Takuya, Matsumoto Yoshihiro

机构信息

Department of Orthopaedic Surgery, Fukushima Medical University School of Medicine, Fukushima, Japan.

Higashi-Shirakawa Orthopaedic Academy, Fukushima Medical University School of Medicine, Fukushima, Japan.

出版信息

Case Rep Oncol. 2025 Jun 16;18(1):972-978. doi: 10.1159/000546985. eCollection 2025 Jan-Dec.

Abstract

INTRODUCTION

Giant cell tumors of bone (GCTBs) are locally aggressive tumors that can induce pathological fractures. Previous reports suggest that denosumab induces intratumoral ossification in β-catenin positive GCTB. However, its effect on the complete resection of GCTB with extraskeletal lesions remains unclear.

CASE PRESENTATION

A 45-year-old woman with a pathological fracture of the left distal femur was diagnosed with GCTB based on the biopsy findings, which revealed both H3.3 G34W and nuclear β-catenin positivity. Eight courses of neoadjuvant denosumab therapy were administered to visualize the extraskeletal lesions; subsequently, en bloc resection with megaprosthetic reconstruction was successfully performed. Pathological examination revealed intratumoral ossification without osteoclastic giant cells. Two years after the surgery, no evidence of local recurrence was recorded.

CONCLUSION

Neoadjuvant denosumab therapy is an effective approach for managing β-catenin positive GCTB with pathological fractures as it promotes intratumoral ossification and facilitates complete resection of GCTB, including extraskeletal lesions.

摘要

引言

骨巨细胞瘤(GCTB)是具有局部侵袭性的肿瘤,可导致病理性骨折。既往报道显示,地诺单抗可诱导β-连环蛋白阳性的GCTB发生瘤内骨化。然而,其对伴有骨外病变的GCTB完整切除的影响尚不清楚。

病例介绍

一名45岁女性,左股骨远端病理性骨折,活检结果显示H3.3 G34W和核β-连环蛋白均呈阳性,据此诊断为GCTB。给予8个疗程的新辅助地诺单抗治疗以明确骨外病变;随后,成功进行了大块切除及假体置换重建。病理检查显示瘤内骨化,无破骨巨细胞。术后两年,未记录到局部复发的证据。

结论

新辅助地诺单抗治疗是一种有效的治疗方法,可用于治疗伴有病理性骨折的β-连环蛋白阳性GCTB,因为它可促进瘤内骨化并有助于GCTB的完整切除,包括骨外病变。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/0e4c/12266702/82c6787d1748/cro-2025-0018-0001-546985_F01.jpg

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